Causes of double-negative T-cell lymphocytosis in children and adults

J Clin Pathol. 2020 Jul;73(7):431-438. doi: 10.1136/jclinpath-2019-206255. Epub 2019 Dec 6.

Abstract

Aims: The causes and diagnosis of 'double-negative' (CD3+CD4-CD8-) T-cell lymphocytosis are not well studied. We aimed to define the causes of double-negative T-cell lymphocytosis in children and adults, and to identify simple clinical and laboratory features that would help to differentiate between the underlying conditions.

Methods: We collected clinical and laboratory data on 10 children and 30 adults with significantly increased peripheral-blood double-negative T-cells (>10% of total lymphocytes). We identified conditions associated with double-negative T-lymphocytosis with flow cytometry, peripheral-blood morphology and T-cell receptor-gene rearrangement studies. Patients were assigned to diagnostic categories on the basis of these test results.

Results and conclusions: The causes of double-negative T-cell lymphocytosis in children were autoimmune lymphoproliferative syndrome (ALPS) and reactive γ/δ Τ-lymphocytosis. T-cell large granular lymphocyte (T-LGL) leukaemia, reactive γ/δ T-lymphocytosis and hepatosplenic T-cell lymphoma (HSTL) were the the most common disorders underlying double-negative T-cell lymphocytosis in adults. Less common causes included hypereosinophilic syndrome, peripheral T-cell lymphoma, ALPS and monoclonal, double-negative T-lymphocytosis of uncertain significance. CD5/CD7/Vδ2 expression and absolute double-negative lymphocyte count (<1.8×109/L) were useful discriminators for distinguishing patients with reactive γ/δ T-lymphocytosis from those with γ/δ lymphoproliferative disorders. Differentiating between γ/δ T-LGL and HSTL can be difficult. Expression of CD57 and cellular morphology (pale cytoplasm with distinct granules) would support a diagnosis of γ/δ T-LGL.

Keywords: flow cytometry; haemato-oncology; haematology; haematopathology; morphology.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Autoimmune Lymphoproliferative Syndrome / complications*
  • CD4 Antigens / immunology
  • CD57 Antigens / immunology
  • CD8 Antigens / immunology
  • Child
  • Child, Preschool
  • Female
  • Greece
  • Humans
  • Leukemia, Large Granular Lymphocytic / complications*
  • Lymphocyte Count
  • Lymphocytosis / diagnosis*
  • Lymphocytosis / etiology
  • Lymphocytosis / pathology
  • Lymphoproliferative Disorders / complications*
  • Male
  • Middle Aged
  • Retrospective Studies
  • Young Adult

Substances

  • CD4 Antigens
  • CD57 Antigens
  • CD8 Antigens